RESEARCH ARTICLE
Leprosy in children under 15 years of age in
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OPEN ACCESS
Citation: Vieira MCA, Nery JS, Paixão ES, Freitas
de Andrade KV, Oliveira Penna G, Teixeira MG
(2018) Leprosy in children under 15 years of age in
Brazil: A systematic review of the literature. PLoS
Negl Trop Dis 12(10): e0006788. https://doi.org/
10.1371/journal.pntd.0006788
Editor: Carlos Franco-Paredes, Hospital Infantil de
Mexico Federico Gomez, UNITED STATES
Received: April 16, 2018
Accepted: August 24, 2018
Published: October 2, 2018
Copyright: © 2018 Vieira et al. This is an open
access article distributed under the terms of the
Creative Commons Attribution License, which
permits unrestricted use, distribution, and
reproduction in any medium, provided the original
author and source are credited.
Data Availability Statement: All relevant data are
within the paper and its Supporting Information
files.
Funding: This study was supported by Medical
Research Council (MRC) (MR/N017250/1 to L.C.
R.) and CONFAP/ESRC/MRC/BBSRC/FAPDF 2015
– Doenças Negligenciadas (Processo FAP-DF
193.000.008/2016 to G.O.P). MCAV scholarship
was provided by Fundação de Amparo à Pesquisa
do Estado da Bahia (FAPESB). The funders had no
role in study design, data collection and analysis,
PLOS Neglected Tropical Diseases | https://doi.org/10.1371/journal.pntd.0006788 October 2, 2018
Brazil: A systematic review of the literature
Michelle Christini Araújo Vieira ID1*, Joilda Silva Nery ID2, Enny S. Paixão ID3, Kaio
Vinicius Freitas de Andrade ID2,4, Gerson Oliveira Penna ID5,6, Maria Glória Teixeira ID2
1 Nursing School, Federal University of the Vale do São Francisco, Petrolina, Pernambuco, Brazil, 2 Institute
of Collective Health, Federal University of Bahia, UFBA, Salvador, Brazil, 3 Department of Infectious Disease
Epidemiology, Faculty of Epidemiology and Population Health, London School of Hygiene & Tropical
Medicine, London, United Kingdom, 4 Health Departament, State University of Feira de Santana, Feira de
Santana, Bahia, Brazil, 5 Tropical Medicine Center, Brası́lia University, Brası́lia, Distrito Federal, Brazil,
6 School of Government of the Oswaldo Cruz Foundation, Brazilian Ministry of Health, Brası́lia, Distrito
Federal, Brazil
* michelle.christini@gmail.com
Abstract
Background
Leprosy is a chronic infectious disease neglected, caused by Mycobacterium leprae, consid-
ered a public health problem because may cause permanent physical disabilities and defor-
mities, leading to severe limitations. This review presents an overview of the results of
epidemiological studies on leprosy occurrence in childhood in Brazil, aiming to alert health
planners and managers to the actual need to institute special control strategies.
Methodology/Principal findings
Data collection consisted of an electronic search for publications in eight databases: Litera-
tura Latino-Americana e do Caribe em Ciências da Saúde (LILACS), Scientific Electronic
Library Online (SciELO), PuBMed, Biblioteca Virtual em Saúde (BVS), SciVerse Scopus
(Scopus), CAPES theses database, CAPES journals database and Web of Science of
papers published up to 2016. After apply selection criteria, twenty-two papers of studies
conducted in four different regions of Brazil and published between 2001 and 2016 were
included in the review. The leprosy detection rate ranged from 10.9 to 78.4 per 100,000
inhabitants. Despite affecting both sexes, leprosy was more common in boys and in 10-14-
year-olds. Although the authors reported a high cure proportion (82–90%), between 1.7%
and 5.5% of the individuals developed a disability resulting from the disease.
Conclusions/Significance
The findings of this review shows that leprosy situation in Brazilian children under 15 years
is extremely adverse in that the leprosy detection rate remains high in the majority of studies.
The proportion of cases involving disability is also high and reflects the difficulties and the
poor effectiveness of actions aimed at controlling the disease. The authors suggest the
development of studies in spatial clusters of leprosy, where beyond the routine actions
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 Leprosy in children under 15 years of age in Brazil

decision to publish, or preparation of the established, are included news strategies of active search and campaigns and actions of
manuscript. All authors had full access to the educations inside the clusters of this disease. The new agenda needs to involve the pre-
review data and share final responsibility for the
decision to submit for publication.
cepts of ethical, humane and supportive care, in order to achieve a new level of leprosy con-
trol in Brazil.
Competing interests: The authors have declared
that no competing interests exist.

Author summary
Leprosy remains as a severe health problem in Brazil and its transmission in children
under 15 years of age occurs mainly through intradomiciliary contacts. The number of
leprosy cases in this age group is considered an important indicator for the surveillance of
this disease. To understand how the epidemiological studies in Brazil have shown the situ-
ation of young people affected by leprosy, we performed a systematic review of the litera-
ture, searching for published articles about the situation of leprosy in this age group. We
reviewed 22 studies published during 2001 to 2016 and concluded that the negative effects
of leprosy still remain high in most of studied places in Brazil. This disease was more com-
mon in boys, aged between 10 to 14 years old, with a remarkable proportion of disabilities
due to leprosy. These disabilities can limit their routine activities and reflect failure in the
public medical care. We hope that our review should contribute to arguments in order to
improve the control of this disease in children.

Introduction
Leprosy is a chronic infectious disease caused by Mycobacterium leprae. Since the disease may
cause permanent physical disabilities and deformities, leading to severe limitations in individ-
ual’s ability to perform daily activities, this disease is considered a public health problem
worldwide [1, 2]. The incubation period of M. leprae is very long, in some cases up to ten
years, and for this reason the majority of cases only become clinically detectable in adulthood.
The occurrence of leprosy in children under 15 years of age suggests early exposure and persis-
tent transmission of the agent [3].
Leprosy control has improved markedly around the world over the past thirty years. During
this period the leprosy prevalence fell from 21.1 per 10,000 inhabitants in 1983 to 0.24 per
10,000 inhabitants in 2000. This decline occurred due to the generalized use of multidrug ther-
apy (MDT), in addition to nationwide campaigns and an improvement in the quality of health
services directed to leprosy treatment in endemic countries [1]. In 2016, the World Health
Organization (WHO) launched a new global strategy entitled “The Global Leprosy Strategy
2016–2020: Accelerating towards a leprosy-free world” [4].
The total number leprosy new cases registered by the WHO in 2016 was 214,783 (2.9 per
100,000 inhabitants), 95% of which are concentrated in only 14 countries of high endemicity
where over 1,000 new leprosy cases are notified each year [1]. These countries are geographi-
cally situated within the tropics, with India in first place and Brazil coming second in terms of
the number of cases detected annually.
In 2017, the total number of leprosy new cases in Brazil was 22,940, of which 1,718 were in
children under 15 years of age, corresponding to 7.5% and detection rate of 3.72 cases per
100,000 inhabitants [5]. One of the goals of the Global Leprosy Strategy 2016–2020 is to further
reduce global and local leprosy burden, aiming to reduce to zero the number of children with
disabilities due to this disease. However, in the last five years in Brazil, the percentage of

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 Leprosy in children under 15 years of age in Brazil

children <15 years old that has grade 2 disability (G2D) ranged from 2.9% (2013) to 4.1%
(2017), with an average proportion of 3.7%, reflecting long delay in diagnosis [5].
One of characteristics of the distribution of leprosy is the occurrence in clusters, and in Bra-
zil the leprosy detection rate in the all population and in the under 15 years of age, varies
greatly among regions and cities. A region is considered hyperendemic when the leprosy
detection rate in under 15 years of age is above 10 per 100,000 inhabitants [6], in many areas,
the values of this indicator reach higher levels those considered hyperendemic [5].
In 2016, the detection rate of new cases in this country reached 12.2 per 100,000 inhabi-
tants, considered “high” according to the reference parameters established by the Ministry of
Health [5]. In the northern region, this indicator in the general population was 28.7 per
100,000 inhabitants, and of 8.92 per 100,000 inhabitants under 15 years of age. In the Midwest,
30.01 new cases per 100,000 inhabitants were registered for the general population and 6.42
per 100,000 inhabitants under 15 years of age. In the Northeast of the country, the detection
rate in the general population was 19.29 per 100,000 inhabitants, while in those under 15 years
of age was 5.78 per 100,000 inhabitants [7, 8].
Based on fact that leprosy in children under 15 years of age is hiperendemic in Brazil and
that produce important negative effect on life of the affected children and their families, the
research question that guided this systematic review was: Does the epidemiological studies on
leprosy in childhood, in Brazil, have warning to the policy makers and health managers that
the severity of negative effects of this disease is demanding immediate attention? This review
presents an overview of the results of epidemiological studies on leprosy occurrence in chil-
dren under 15 years, in Brazil, aiming to alert health planners and managers to the need to
institute special control strategies for this disease, which is one of the most neglected problems
of public health.

Methods
This study was conducted according to the guidelines established in the Preferred Reporting
Items for Systematic Reviews and Meta-Analyses [9], with the study protocol registered in
PROSPERO with reference code CRD42016033006PROSPERO.

Selection criteria and search

The review was conducted between May 17 and August 10, 2016 using the databases listed in
Table 1. Equivalent keywords or subject terms were identified using health sciences descriptors
(DeCS), a trilingual structured vocabulary created by the Latin American and Caribbean Cen-
ter on Health Sciences Information (LILACS/BIREME) [10], in the three languages (Portu-
guese, English and Spanish) included in this review. A simulated search by language was then
conducted to verify which terms would achieve the optimal results in each database. The
appropriateness of the language used in the search was assured by preserving the three lan-
guages as filters in the databases.
Although the term “children under 15 years of age” was used as a keyword in various stud-
ies conducted in Brazil and internationally, no such equivalent was found in the DeCS, with
“minors” being the closest term found. Therefore, in addition to the terms child and adolescent,
the term “children under 15 years of age” was also used. Likewise, after a preliminary reading of
the international papers, it was found that two forms were used in international papers when
referring to “children under 15 years of age”: “under 15 years” and “younger than 15 years”.
Therefore, the following search terms were used and restricted to the fields “title”, “abstract”
and “keywords”:

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 Leprosy in children under 15 years of age in Brazil

Table 1. Details of the database search procedure.

Database Website Date
CAPES theses database http://bancodeteses.capes.gov.br/ 10/Aug/
2016
Biblioteca Virtual em Saúde–(BVS) http://brasil.bvs.br/ 17/May/
2016
Literatura Latino-Americana e do Caribe http://lilacs.bvsalud.org/ 17/May/
em Ciências da Saúde (LILACS) 2016
CAPES Journals http://www-periodicos-capes-gov-br.ez21.periodicos.capes.gov.br/index.php?option=com_phome 28/July/
2016
PubMed http://www.ncbi.nlm.nih.gov/pubmed 17/May/
2016
Scientific Electronic Library Online http://scielo.org/php/index.php 05/June/
(SciELO) 2016
SciVerse Scopus (Scopus) https://www.scopus.com/ 07/June/
2016
Web of Science https://apps.webofknowledge.com/UA_GeneralSearch_input.do?product=UA&search_mode= 06/June/
GeneralSearch&SID=3D42qcvkZTMW85yA9LR&preferencesSaved = 2016
https://doi.org/10.1371/journal.pntd.0006788.t001

Brazilian databases: BVS, CAPES theses database and Scielo: Hansenı́ase AND “Epide-
miologia” AND “Criança”; Hansenı́ase AND “Epidemiologia” AND “Adolescente”; “Hanse-
nı́ase AND “Epidemiologia” AND “Menores de 15 anos”.
International databases: LILACS, CAPES journals, PubMed, Scopus, Web of Science:
"Leprosy" AND "Epidemiology" AND “Child”, "Leprosy" AND "Epidemiology" AND “Adoles-
cent”, "Leprosy" AND "Epidemiology" AND “Under 15 years” OR “Younger than 15 years”,
“Lepra” AND “Epidemiologı́a” AND “Niño”, “Lepra” AND “Epidemiologı́a” AND “Adoles-
cente”, “Lepra” AND “Epidemiologı́a” AND “menor de 15 años”.
The protocol defined the following inclusion criteria: complete papers and theses available
in the selected databases in English, Portuguese or Spanish. Since there were few scientific
papers specifically on leprosy in children under 15 years of age, a decision was made not to
restrict the beginning of the search period to any specific year, thereby including all the arti-
cles, dissertations and theses found up to the cut-off date of August 2016, in which the type of
epidemiological study was described in the Methods section.
The exclusion criteria consisted of papers involving animals; articles published as a short
communication or poster; papers in which the methodology used was not described; any other
type of documents; and duplicates.
The data extracted from the selected papers were transferred to an Excel 6.0 version spread-
sheet using double data entry and evaluated independently by two reviewers (MCAV and
KVFA). All disagreements were settled by a third reviewer (MGT).
The relevant data that were collected in the publications selected for the systematic review
were: authors, year of publication, journal, study site, study period, study design, population,
age group, sex, education, type of housing, leprosy detection rate, epidemiological characteri-
zation, operational classification, clinical classification, vaccination with Calmette-Guérin
Bacillus (BCG) scar, smear test result, disability degree at diagnosis, disability degree at cure,
Hansen’s reaction, close contact with leprosy patient, contact examination, 2nd BCG in con-
tacts, detection mode, reason for discharge, complete treatment, relapse and sequelae.

Evaluation of the quality of studies

A scale was created to evaluate the quality of the articles selected for this systematic review (S1
Table). This scale was based on and adjusted according to the proposals made by Downs and

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 Leprosy in children under 15 years of age in Brazil

Black [11] and Boas and Neto [12]. Scores were awarded for the selected items on a 17-point
scale and analyzed according to the mean score awarded for the quality of the articles.

Results
A total of 2,075 papers were identified in the databases following a search based on the selected
keywords. Subsequently, 1,880 articles were excluded because: they did not meet the study
objectives, they dealt with subject matter other than that proposed, duplicates, the papers were
not available in their entirety, the age group included in the study was older than that estab-
lished for this review, or the objective of the study was not included in the keywords, title and/
or abstract. Fig 1 consists of a flowchart depicting the selection of the articles in accordance
with the PRISMA guidelines.
The 22 studies selected for inclusion [3, 13–33] were published between 2001 and 2016, and
the most productive year was 2010 (04 papers). The predominant database was the BVS from
which eight papers were retrieved and included in the review. Geographically, the papers iden-
tified came from four regions of the country: the Northeast, Southeast, Midwest and North, as
described in Table 2.
All studies included in this systematic review were based on scientific research (n = 22).
Most of the studies were linked to universities (n = 20) and only two were from the Brazilian
Ministry of Health. The studies were financed by the authors’ own resources (n = 14), by the
National Scientific and Technological Development—CNPq (n = 06) and Ministry of Health
(n = 02).
S2 Table 2 describes the papers in greater detail: author, year of publication, type of study,
study population, study period, objective, main findings and score according to the methodol-
ogy validation scale. The studies were ecological, cross-sectional or case series descriptive.
Sample sizes ranged from 24 to 2,455 individuals of both sexes. Most of the studies provided
descriptions of the participants according to age (0–4 years, 5–9 years and 10–14 years), sex,
detection rate, epidemiological classification, operational classification, clinical classification
and degree of disability. Of the 14 publications in which leprosy was presented proportionally
by sex, males were more affected in 8. With respect to the distribution of cases according to
age group, the greatest number of cases occurred in 10-14-year-olds (n = 10). Only 13 papers
provided the clinical classification of the disease and in 6 of these articles the most common
was tuberculoid leprosy. Operational classification was described in 16 studies, with the pauci-
bacillary form being predominant in 14. The classification of the epidemiological situation of
the studies place was described in 17 articles, with 11 reporting hyperendemic levels in chil-
dren under 15 years of age, with detection rates that ranged from 10.9 to 78.4 per 100,000
inhabitants. Of these 19 papers, 5 reported prevalence in the states of Alagoas, Minas Gerais,
Mato Grosso, Pará and Rio de Janeiro, with the level being highest in Mato Grosso at 25.9 per
100,000 inhabitants in 2014.
The proportion of cases diagnosed with grade 2 disability (n = 13) ranged from 1.7% to
5.5%. However, the percentage of individuals in this group who had not undergone evaluation
(n = 5) ranged from 2.4% to 19.20%. Only five articles reported the proportion of grade 2 dis-
abilities following cure (ranging from 0.5% to 11.1%) and only three reported on the propor-
tion of the sample that was not evaluated, citing percentages that ranged from 18% to 61.5%.
Household contact, with the family being the main source of transmission, was reported in
three publications. According to the reports, 40% of the children had parents with leprosy, 20–
36% a grandparent, 18% an uncle or aunt, and 4% had siblings with the disease.
The examination of contacts was reported in four publications, with 24.2% to 90% of con-
tacts having been examined. The principal form of detection reported (n = 8) was through

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 Leprosy in children under 15 years of age in Brazil

Fig 1. PRISMA flow diagram for the systematic review. Accordingly, the titles and abstracts of 195 publications were evaluated. Fifty-nine of these papers met
the inclusion criteria and were then selected to be read in their entirety. After the three reviewers had read these publications, 22 studies (articles, theses and
dissertations) were selected for inclusion in this review (Table 2).
https://doi.org/10.1371/journal.pntd.0006788.g001

spontaneous demand at healthcare facilities (13.8% to 79.6%), followed by referral from

another healthcare service (12.4% to 75.6%). Other forms of detection such as collective exami-
nations (n = 6) and the examination of contacts (n = 7) were also reported, with percentages
ranging from 1% to 6.6% and from 8.9% to 20.4%, respectively.
The presence of a BCG vaccination scar and of a second dose of this vaccine for the contacts
was described in only two studies, with results showing that the presence of a scar from the

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Table 2. Details of the papers selected, including author, year, title, journal and database.
Ref Author/Year
BRAZIL
[13] Levantezi, 2014
Northeast
[14] Alencar et al., 2008
[15] Bastos, Raquel Patriota
Cota
2010
[16] Luna et al., 2013
[17] Moreira et al., 2014
[18] Oliveira, Carlos Alberto
Rodrigues de, 2008
[19] Santos et al., 2015
[20] Santos et al., 2016
[21] Souza et al., 2001
[22] Souza et al., 2012
Southeast
[23] Araújo et al., 2004
[24] Ferreira et al., 2005
[25] Ferreira, Maria Aparecida
Alves
2012
[26] Flach et al., 2010
[3] Lana et al., 2007
[27] Morais et al., 2010
Midwest
[28] Freitas, Bruna Hinnah
Borges Martins de
2015
North
[29] Barreto et al., 2012
[30] Coelho Júnior, José Maria
dos Santos, 2010
[31] Franco, Mariane Cordeiro Dinâmica de transmissão da hansenı́ase em menores de 15 anos em área
Alves, 2014 hiperendêmica na região Norte do Brasil
[32] Guimarães, Andréa De
Cassia Lima, 2011
[33] Imbiriba et al., 2008
https://doi.org/10.1371/journal.pntd.0006788.t002
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Leprosy in children under 15 years of age in Brazil
Title Journal Database
Leprosy in children under fifteen years in Brazil, 2011 Lepr Rev CAPES Journals
Hansenı́ase no municı́pio de Fortaleza, CE, Brasil: aspectos Rev Bras Enferm Web of Science
epidemiológicos e operacionais em menores de 15 anos (1995–2006)
Hansenı́ase: estudo epidemiológico e clı́nico dos casos ocorridos em UFAL thesis CAPES database of theses
menores de 15 anos no estado de Alagoas, no perı́odo de 1990–2007
Perfil clı́nico-epidemiológico da hansenı́ase em menores de quinze anos Rev Bras Promoc Saúde Biblioteca Virtual em
no municı́pio de Juazeiro-BA Saúde–(BVS)
Epidemiological situation of leprosy in Salvador from 2001 to 2009 An Bras Dermatol Web of Science
Perfil Epidemiológico da Hansenı́ase em Menores de 15 Anos no Dissertation ENSP Biblioteca Virtual em
Municı́pio de Teresina Saúde–(BVS)
Leprosy and Disability in Children Younger Than 15 Years in an The Pediatric Infectious Biblioteca Virtual em
Endemic Area of Northeast Brazil Disease Journal Saúde–(BVS)
Leprosy in children and adolescents under 15 years old in an urban Mem Inst Oswaldo Cruz SciVerse Scopus (Scopus)
centre in Brazil
Aplicação de modelo bayesiano empı́rico na análise espacial da Rev Saúde Pública Biblioteca Virtual em
ocorrência de hansenı́ase Saúde–(BVS)
Epidemiological Profile of Leprosy in the Brazilian state of Piauı́ An Bras Dermatol Biblioteca Virtual em
between 2003 and 2008 Saúde–(BVS)
Detecção da hansenı́ase na faixa etária de 0 a 14 anos em Belo Horizonte Rev Med Minas Gerais Biblioteca Virtual em
no perı́odo 1992–1999: implicações para o controle Saúde–(BVS)
Hansenı́ase em menores de quinze anos no municı́pio de Paracatu, MG Rev Bras Epidemiol Scientific Electronic
(1994 a 2001) Library Online (SciELO)
Evolução das taxas de detecção de casos de hansenı́ase em menores de 15 UFMG Thesis CAPES database of theses
anos no estado de Minas Gerais de 2001 a 2010
Análise da série histórica do perı́odo de 2001 a 2009 dos casos de Hansen Int Biblioteca Virtual em
hansenı́ase em menores de 15 anos, no Estado do RJ Saúde–(BVS)
Hansenı́ase em menores de 15 anos no Vale do Jequitinhonha, Minas Rev Bras Enferm Web of Science
Gerais, Brasil
Avaliação das ações de controle da hansenı́ase no municı́pio de Hansen Int Biblioteca Virtual em
Governador Valadares, Brasil, no perı́odo de 2001 a 2006 Saúde–(BVS)
Indicadores e determinantes clı́nicos e epidemiológicos de hansenı́ase Dissertation UFMG CAPES database of theses
em menores de quinze anos, Mato Grosso, Brasil
High rates of undiagnosed leprosy and subclinical infection amongst Mem Inst Oswaldo Cruz Web of Science
school children in the Amazon Region
Tendência da incidência de hansenı́ase em menores de 15 anos em Dissertation UFPA CAPES database of theses
Jacundá-Pará e sua relação com a implantação dos serviços de saúde
Thesis UFPA CAPES database of theses
Prevalência de incapacidades sensitivomotoras por doença hansênica em Dissertation UFPA CAPES database of theses
pacientes menores de 15 anos
Perfil epidemiológico da hansenı́ase em menores de quinze anos de Rev Saúde Pública Web of Science
idade, Manaus (AM), 1998–2005
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 Leprosy in children under 15 years of age in Brazil

first dose of this vaccine was high; however, revaccination of contacts was very low, between
13.8% and 15.5%.
Five studies reported on acid-fast microscopy results, with this test having been performed
in 20.6% to 94.6% of cases and with positivity ranging from 4.3% to 15%. In 4.4% to 79.3% of
cases, this test was not performed.
Only two papers described the occurrence of a leprosy reaction. In one of these articles,
reaction occurred in 33.4% of patients: immediately following diagnosis in 24.4% and follow-
ing cure in 9%. The other study simply mentioned that 44.1% of the patients did not develop a
leprosy reaction.
Complete treatment involving six doses of multidrug therapy (MDT) was administered in
41.7% to 66.7% of cases, with 12 doses of MDT being administered in 11.8% to 45.8% of cases.
In another 2.14% to 21.5% of cases, treatment regimens were longer or unknown. Recurrences
were recorded in 3.4% of children. Complete cure ranged from 81.9% to 90% of cases. Few
studies emphasized the importance of drawing the attention of health planners and managers
to the need to develop special actions for childhood.

Discussion
This systematic review included 22 articles and showed that the detection rate of leprosy in
children under 15 years of age, in Brazil, comes decreasing, but it remains very high in the
most of sites investigated reaching levels compatible with hyperendemicity, or at least high or
moderate endemicity. It was shown strong association between poverty and leprosy and that
its spatial distribution is in clusters. The boys were the most affected by this disease; the pro-
portion of multibacillary cases was high (23.4% to 75%) considering that they were children;
the proportion of cases with grade 2 disability varied in the different studies analyzed, from
moderate (1.7% to 5.5%) to high at the time of bacteriological cure (18% to 61.5%)and; the pro-
portion of reaction leprosy was also high (33.4% to 55.1%). The coverage of intradomiciliary
contacts exams did not reach satisfactory levels, as well as was very low the coverage of the sec-
ond dose BCG.
These findings clearly indicate that the socio environmental conditions for determination
of leprosy occurrence in Brazil are maintained, despite efforts, which have been undertaken to
eliminate it, in the last three decades, when there was prevalence reduction near 94%, in total
population. These efforts has made possible the continuous reduction in the prevalence and
leprosy detection rate in adults and in children in this country as a whole [34], but has not
been able to avoid the continued transmission and expansion of this disease that persist in the
clusters, particularly in the Midwest, North and Northeast regions [35]. Such situation shows
the early contamination of young people, consequent to the failure to detect and treat timely
adults bacilliferous cases who maintains the chain of transmission in communities [20]. Luna
et al. [16] and Santos et al [20] demonstrate that the distribution of new cases of leprosy differs
from area to area, and highlight the need to sensitize managers to perform actual diagnosis by
region, with actions being prioritized according to the epidemiological profile. In this perspec-
tive, the implementation of health education measures would theoretically bring benefits,
mainly in the major cities, where leprosy has been found concentrated in bounded clusters.
The fact that children with leprosy were mostly from families living in great social vulnera-
bility related to low socioeconomic conditions [24, 25, 32] is not a new or unknown fact. It is
not by chance, that some of the studies included here reveal that these children have more dif-
ficulties in progressing in schooling, that will result in the future in perpetuating poverty, simi-
lar to their parents. This is not determined by the physical damages that the disease causes, but
by the disease stigma and insufficient social protection. Unfortunately, this situation has been

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 Leprosy in children under 15 years of age in Brazil

maintained until the present day as more recent studies show. For example, it was found a sta-
tistically significant association between greater coverage by the conditional cash transfer pro-
gram Bolsa Família and reduction in the detection rates of leprosy. The authors argue that, in
view of its characteristics, this social protection strategy is capable of acting on different social
determinants of leprosy such as poverty and economic inequality [36, 37].
As Brazil´s Leprosy Control Programme is integrated in the primary health care while in
other countries leprosy interventions are developed in vertical programmes, it is difficult to
establish comparisons among endemic countries. Although in Brazil the actions of this Pro-
gramme are implemented in the whole network of public health services one of difficulties
faced is that the majority of new cases reported are known from passive detection by these ser-
vices, in which the demand is mostly constituted by poor population. There is evidence that
when active case search is implemented, unexpectedly higher new case detection rates have
been found, even in cities with the highest human development index (HDI), as in São Paulo
[38] and Brası́lia-District Federal [39].
As regards to the higher frequency of boys with leprosy observed in the analyzed studies,
further studies need to be conducted to clarify whether boys are indeed at a greater risk of
acquiring the disease. If confirmed, a hypothesis to explain this fact could be their greater
exposure to social interactions more frequent and intense when compared to girls, increasing
the possibility of contact with people with leprosy outside the dwelling [25].
As we know, the basic strategy for the control of leprosy focuses on eliminating the sources
of infection using MDT. For this strategy to be effective it has to be adopted timely, i.e. as early
as possible to ensure that patients are protected from developing a disability and to reduce the
time of transmissibility of the agent. Nevertheless, the proportions of severe disability found in
this review are clear proof that diagnosis of the disease is being made late, increasing the risk
of nerve damage [26], producing negative repercussion on the daily life of this young.
Undoubtedly, physical disabilities negatively affect child’s development, stigmatizing the indi-
vidual, causing severe psychological repercussions and affecting his/her social life, and it can
also reduce a person’s future ability to enter the job market [14, 26, 33]. On the other hand, the
high rates of leprosy reaction found highlight the need to increase the availability of specialist
services for the prevention of physical disabilities in young people affected by this disease [24].
Not by chance, one of the principal goals of the new Global Leprosy Strategy 2016–2020 [4], is
to eliminate grade-2 disabilities in pediatric patients by implementing quality services aimed
principally at women and children.
In addition, the predominance of tuberculoid form indicates that immunocompetent indi-
viduals also are being affected, highlighting that Mycobacterium leprae is circulating intensely
[3, 17, 24, 40]. Another finding that gives further strength to this affirmative is the proportion
of multibacillary cases which cannot be considered low for the age group under 15 years of
age. On the contrary, these cases are concerning, since means that the young people most sus-
ceptible to the disease are becoming infected at a very early age, representing yet more one evi-
dence that the process of transmission is intense and continuous [21]. The low proportion the
healthy contacts vaccinated, measured by the scar presence of the BCG second dose of the con-
tacts, found in the present review, shows possible failure of surveillance to avoid multibacillary
forms.
The main limitation of this systematic review was the low quantity of studies available in
the scientific literature, highlighting how much leprosy is a neglected disease and, in particular,
when it refers to childhood. In addition, most of these studies were descriptive, using surveil-
lance secondary data that, in general show some inconsistency in relation to the quality and
quantity of information. These limitations did not allow the analysis of individual and collec-
tive risk factors among other epidemiological aspects.

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 Leprosy in children under 15 years of age in Brazil

Conclusions
In spite of these limitations, the results of this study will be useful, especially for public health,
since they will help to alert the decision makers to the need in identifying the care needs of
those under 15 years of age affected by the disease due to social stigma and the leprosy physical
repercussions in the children.
Although it has not been the object of evaluation of most of the studies, several authors
point out that one of the problems of leprosy control is the fragility of surveillance, since the
action of health services is predominantly individualized, with a low proportion of search
active contacts. The detection of new cases depends on the spontaneous demand of the indi-
vidual, who mostly seek the health service in advanced stage of the disease, which increases the
risk of permanent damage. [3, 5, 19, 23].
The presence of the contacts and the communities in the greatest risk of disease, should be
found of the pillars of collective actions, continues being one of the main force to overcome
those in the Brazilian leprosy control program. Besides, the treatment of leprosy patients still
not meet the target established (98%) by Brazilian Ministry of Health and WHO [25].
In view of this adverse scenario, we suggested the development of studies in spatial clusters
of leprosy, where beyond of the control and surveillance actions established by the Brazilian
Ministry of Health, are included news strategies of active search adding social contacts, cam-
paigns and actions of educations inside these areas aiming early diagnoses and treatment. If
many new cases in children will be discovered in these communities, it is understood that the
search must be expanding to others neighboring area to uncover the primary sources of infec-
tion to prevent new cases in childhood.
For this, it is necessary a debate and initiatives needs to be carried out to establish an agenda
aimed at strengthening the active surveillance of this disease. This agenda needs to involve the
precepts of ethical, humane and supportive care in order to achieve a new level of leprosy con-
trol in Brazil.

Supporting information
S1 Table. Methodological validation scale for the articles.
(DOCX)
S2 Table. Articles selected according to the author, year, type of study, population, study
period, objective, principal findings and score according to the methodological validation
scale.
(DOCX)

Author Contributions
Conceptualization: Michelle Christini Araújo Vieira, Joilda Silva Nery, Maria Glória Teixeira.
Data curation: Michelle Christini Araújo Vieira.
Formal analysis: Michelle Christini Araújo Vieira.
Investigation: Michelle Christini Araújo Vieira, Joilda Silva Nery, Kaio Vinicius Freitas de
Andrade.
Methodology: Michelle Christini Araújo Vieira, Joilda Silva Nery, Kaio Vinicius Freitas de
Andrade.
Project administration: Michelle Christini Araújo Vieira.

PLOS Neglected Tropical Diseases | https://doi.org/10.1371/journal.pntd.0006788 October 2, 2018 10 / 13

 Leprosy in children under 15 years of age in Brazil

Resources: Michelle Christini Araújo Vieira.

Software: Michelle Christini Araújo Vieira.
Supervision: Michelle Christini Araújo Vieira, Joilda Silva Nery, Enny S. Paixão, Gerson Oli-
veira Penna, Maria Glória Teixeira.
Validation: Michelle Christini Araújo Vieira, Enny S. Paixão, Gerson Oliveira Penna, Maria
Glória Teixeira.
Visualization: Michelle Christini Araújo Vieira, Maria Glória Teixeira.
Writing – original draft: Michelle Christini Araújo Vieira, Joilda Silva Nery, Enny S. Paixão,
Kaio Vinicius Freitas de Andrade, Maria Glória Teixeira.
Writing – review & editing: Michelle Christini Araújo Vieira, Joilda Silva Nery, Enny S.
Paixão, Kaio Vinicius Freitas de Andrade, Gerson Oliveira Penna, Maria Glória Teixeira.

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